Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Multicenter Study
. 2020 Feb;92(2):159-168.
doi: 10.1111/cen.14128. Epub 2019 Dec 11.

Burden of illness in not adequately controlled chronic hypoparathyroidism: Findings from a 13-country patient and caregiver survey

Heide Siggelkow  1 , Bart L Clarke  2 , John Germak  3 , Claudio Marelli  3 , Kristina Chen  4 , Helen Dahl-Hansen  5 , Elizabeth Glenister  6 , Nawal Bent-Ennakhil  7 , Davneet Judge  7 , Katie Mycock  7 , Jens Bollerslev  8   9
Affiliations
Multicenter Study

Burden of illness in not adequately controlled chronic hypoparathyroidism: Findings from a 13-country patient and caregiver survey

Heide Siggelkow et al. Clin Endocrinol (Oxf). 2020 Feb.

Abstract

Objective: To address knowledge gaps regarding burdens associated with not adequately controlled chronic hypoparathyroidism.

Design: Global patient and caregiver survey.

Study populations: Patients with chronic hypoparathyroidism not adequately controlled on conventional therapy and their caregivers.

Measurements: Health-related quality of life (HRQoL) and health status were evaluated using the 36-item Short Form version 2 (SF-36 v2.0) and Five-Level EuroQoL 5 Dimensions (EQ-5D-5L) instruments, respectively. Hypoparathyroidism-associated symptoms were assessed by a disease-specific Hypoparathyroidism Symptom Diary and caregiver burden via the Modified Caregiver Strain Index (MCSI).

Results: Data were obtained from 398 patients and 207 caregivers. Patients' self-rated hypoparathyroidism-related symptom severity was none (3%), mild (32%), moderate (53%) or severe (12%). Per the Hypoparathyroidism Symptom Diary, patients reported moderate, severe or very severe symptoms of physical fatigue (73%), muscle cramps (55%), heaviness in limbs (55%) and tingling (51%) over a 7-day recall period. Impacts (rated 'somewhat' or 'very much') were reported by 84% of patients for ability to exercise, 78% for sleep, 75% for ability to work and 63% for family relationships. Inverse relationships were observed between patient self-rated overall symptom severity and HRQoL and health status assessment scores-the greater the symptom severity, the lower the SF-36 and EQ-5D-5L scores. Caregiver burden increased with patient self-rated symptom severity: none, 1.7 MCSI; mild, 5.4 MCSI; moderate, 9.5 MCSI; and severe, 12.5 MCSI.

Conclusion: Patients with not adequately controlled hypoparathyroidism reported substantial symptoms and impacts. Greater patient symptom severity was associated with decreased patient HRQoL and health status assessments and increased caregiver burden.

Keywords: caregiver; health status; hypoparathyroidism; parathyroid hormone; patient; quality of life; surveys and questionnaires.

PubMed Disclaimer

Conflict of interest statement

Shire is a member of the Takeda group of companies. HS and JB have served as advisory board members and speakers for Shire. BLC has served as a consultant for Shire. EG is a volunteer with Hypopara UK, which has received donations from Shire. KM is a current employee and DJ and NB‐E are former employees of Adelphi Real‐World, which was contracted by Shire to conduct this research. KC is an employee of Shire Human Genetic Therapies, Inc, a member of the Takeda group of companies. JG and CM are employees of Shire International GmbH, a member of the Takeda group of companies. HD‐H has no conflicts to report.

Figures

Figure 1
Figure 1
(A) EQ‐5D‐5L utility scores* and (B) EQ Visual Analogue Scale scores stratified by patient self‐rated symptom severity. Error bars for (A) represent standard deviation. EQ‐5D‐5L = Five‐Level EuroQoL 5 Dimensions; VAS, visual Analogue scale. *A US scoring algorithm was used for the United States, and an English scoring algorithm was used for Europe and Canada
Figure 2
Figure 2
SF‐36 reported outcomes across domains. SF‐36 = 36‐item Short Form Health Survey
Figure 3
Figure 3
Major impact of hypoparathyroidism on (A) patient relationships categorized by self‐rated hypoparathyroidism symptom severity* (N = 396†) and (B) caregiver relationships (N = 207‡). *None of the patients' self‐rating as having no symptoms of hypoparathyroidism (n = 11) reported a major impact on any personal relationship. †n = 395 for relationships with family, friends and children. ‡n = 206 for relationship with spouse/partner
See this image and copyright information in PMC

References

    1. Bollerslev J, Rejnmark L, Marcocci C, et al. European Society of Endocrinology clinical guideline: treatment of chronic hypoparathyroidism in adults. Eur J Endocrinol. 2015;173:G1‐G120. - PubMed
    1. Brandi ML, Bilezikian JP, Shoback D, et al. Management of hypoparathyroidism: summary statement and guidelines. J Clin Endocrinol Metab. 2016;101:2273‐2283. - PubMed
    1. Shoback DM, Bilezikian JP, Costa AG, et al. Presentation of hypoparathyroidism: etiologies and clinical features. J Clin Endocrinol Metab. 2016;101:2300‐2312. - PubMed
    1. David K, Moyson C, Vanderschueren D, Decallonne B. Long‐term complications in patients with chronic hypoparathyroidism: a cross‐sectional study. Eur J Endocrinol. 2019;180:71‐78. - PubMed
    1. Hadker N, Egan J, Sanders J, Lagast H, Clarke B. Understanding the burden of illness associated with hypoparathyroidism reported among patients in the PARADOX study. Endocr Pract. 2014;20:671‐679. - PubMed

Publication types

MeSH terms